Case Report
Clinical Pathology
Extraskeletal chondroma of the
masseter muscle: a case report
with review of the literature
J. Falleti, R. De Cecio, A. Mentone, V. Lamberti, M. Friscia, S. De Biasi, L. Califano,
L. Insabato: Extraskeletal chondroma of the masseter muscle: a case report with
review of the literature. Int. J. Oral Maxillofac. Surg. 2009; 38: 895–899. # 2009
International Association of Oral and Maxillofacial Surgeons. Published by Elsevier
Ltd. All rights reserved.
J. Falleti1, R. De Cecio1,2, A. Mentone3, V. Lamberti1, M. Friscia3, S. De Biasi3, L. Califano3, L. Insabato1
1Department of Biomorphological and
Functional Science, Section of Anatomic Pathology, Federico II University, Naples, Italy;
2Unit of Anatomic Pathology and
Cytopathology, Regional Oncologic Hospital, CROB, Rionero in Vulture, Potenza, Italy;
3
Department of Maxillofacial Surgery, Federico II University, Naples, Italy
Abstract. The authors report a case of soft tissue chondroma of the masseter muscle in
a 49- year-old man. The tumour was entirely composed of lobules of hyaline
cartilage. The literature on head and neck soft tissue chondroma is also reviewed. To
the authors’ knowledge, this is the first case of muscular soft tissue chondroma in the
head and neck region.
Keywords: soft tissue; cartilaginous; tumour. Accepted for publication 27 February 2009 Available online 5 April 2009
Benign extraosseus cartilaginous tumours are uncommon and usually present as discrete, ossified masses localised in soft tissues. Juxtacortical chondroma, also known as periosteal chondroma, arises from the periosteal region of the long bone or a small bone of the hand or foot30,43. When occurring in extraosseous and synovial locations, it is known as extra-skeletal or soft tissue chondroma12.
Soft tissue chondroma generally affects the soft tissue of the hands and feet26,43in the third and fourth decades of life26; it occasionally occurs in the abdominal wall, and head and neck10. In the head and neck area, the most reported sites are the nasal cavity, paranasal sinuses, larynx10and ton-gue7,24, less frequently, it has been described in the soft palate13, tonsil46, mas-ticatory space10, parotid gland3,18,22 and cheek4,16,18,26. Uncommon sites are the fallopian tubes15, ovaries29and lungs27.
The authors describe a soft tissue chondroma of the left masseter muscle
in a 49- year-old man. To the authors’ knowledge, this is the first case of chon-droma reported at this site.
Case report
A 49-year-old man was admitted with a 6 year history of swelling in the left pre-auricular region that had increased in the last year. Clinical examination revealed a 5 cm diameter, well-circum-scribed nodule, covered by normal skin in the left preauricular region (Fig. 1A); it appeared firm and painless, and floating in superficial planes.
Ultrasound examination revealed a well-circumscribed, 4 cm diameter, nodu-lar mass in the masseter muscle with a non homogeneous hypoechoic structure and calcifications (Fig. 1B).
A computed tomography (CT) scan confirmed the presence of a non homo-geneous density nodule in the left masseter muscle (Fig. 1C). Fine-needle aspiration
cytology of the mass was performed sug-gesting a diagnosis of mixed tumour of the parotid gland. The lesion was surgically excised. A ‘face lift’ cutaneous incision was made in the parotid region and the common trunk of the facial nerve was isolated. Myotomy of the masseter muscle was carried out, and the mass was re-moved (Fig. 1D). The parotid gland was returned to its original position.
The specimen was a 4 cm, well-circum-scribed nodular mass (Fig. 2A). The cut surface was thickened and translucent with a central calcified area (Fig. 2B).
Histological examination showed a well-circumscribed, lobulated mass com-posed of nodules of hyaline cartilage sur-rounded by condensed, collagenous tissue (Fig. 2C). There was variation in cellular-ity within the lesion and the chondrocytic lacunae varied in size (Fig. 2D). Mitotic figures were not seen and there was no evidence of atypia, necrosis or vascular invasion. The tumour was entirely
Falleti et al.
Fig. 2. (A) Gross appearance of the lesion showing a mass with an irregular outer surface and translucent cut section (B). (C) Lobules of hyaline mature cartilage surrounded by a thin fibrous capsule (H/E, 10x). (D) At high power, chondrocytic lacunae with mild atypia are evident (H/E, 20x). Fig. 1. (A) Clinical image showing swelling in the left preauricular region. (B) Ultrasound reveals a 4 cm intramuscular hypoechoic mass with calcifications. (C) CT image demonstrates a tumour mass in the left masseter muscle. (D) Intraoperative image shows a tumour within the masseter muscle.
cessed and serially sectioned but no epithelial components were observed. Immunohistochemically, the tumour cells were strongly positive for S-100 protein, and negative for smooth muscle and myoepithelial markers.
Discussion
The authors report a rare case of a primary benign cartilaginous tumour in the mass-eter muscle of a 49- year-old male of 6 years’ duration. Extraskeletal chondromas are benign tumours arising in soft tissues unrelated to the bone.
The aetiology of soft tissue chondro-mas is unknown but it is thought that they arise from residual embryonic tissue or from metaplastic pluripotential mesench-ymal cells10,39. The tumour cells probably arise from uncommitted mesenchymal stem cells either by metaplastic or neo-plastic processes. Theories about their origin vary, YASUOKAet al. suggested that
tongue chondromas develop from resi-dual embryonal tissue in an area of fetal cartilage, or from pluripotent
mesenchy-mal cells that undergo metaplasia and differentiate into cartilage as a result of an irritating stimulus15,48. In the fallopian tube, the tumour could develop from the mesenchyme of the myosalpinx or subcoelomic mesenchyme of the tubal serosa38.Chondromas and cartilaginous tumours have also been studied cytogen-etically6,40.
Chromosomal alterations are not ran-dom, but can be associated with specific tumour types and their location. Tallini et al. have found cytogenetic similarities and common occurrence of 12q13-15 or +5 alterations in synovial/parosteal or soft tissue chondromas strongly supporting the hypothesis of a common origin for all these lesions40. Abnormalities of chromo-somes 5, 6, 7 and 12, and of chromosomal regions 6q13, 12q13 and 17p13 are shared by malignant and benign cartilaginous tumours6.
The masseter muscle is located in the masticatory space. Chondromas arising in the masseter muscle are difficult to recog-nize clinically and can be mistaken for salivary gland tumours17.
Radiologically, soft tissue chondromas can show irregular calcification without involvement of underlying bone. The most common pattern of calcification is curvi-linear, as in the present case15.
The present case was clinically and cyto-logically mistaken for a salivary gland tumour, although histologically a clear diagnosis of cartilaginous tumour was made. Even serially sectioned epithelial cells were not recognized. Although appearing benign, a full skeletal survey must be carried out to exclude a well-differentiated skeletal chondrosarcoma, which occasionally metastasises to skin. Well-differentiated extraskeletal chon-drosarcoma is rare17. Ectomesenchymal chondromyxoid tumour2,25 must also be considered. It is most often described in the oral cavity2, particularly in the tongue2 but also in the hard palate25. It is a well-circumscribed, but unencapsulated, lesion with a lobular growth pattern and various degrees of cellularity setting in a myxoid, chondroid or hyalinized background. Immunohistochemistry is useful because tumour cells are positive for antibodies Table 1. Review of literature on soft tissue chondroma of the head and neck.
Author Age Sex Location Duration of disease Dimension (cm) IHC
Bruce 19535 NA NA Tongue NA NA NA
Rosen 196131 NA NA Tongue NA NA NA
Yoel 196549 NA NA Tongue NA NA NA
Ramachandran 196828 NA NA Tongue NA NA NA
Hankey 196816 60 F Cheek 31 years 4x4x3 NA
Huppertz 196918 26 F Cheek 2 years 8x5x3 NA
Viglioglia 197044 NA NA Tongue NA NA NA
Samant 197132 NA NA Tongue NA NA NA
Gutmann 197414 NA NA Tongue NA NA NA
Zegarelli 197750 NA NA tongue NA NA NA
del Rio 197811 NA NA Tongue NA NA NA
Jungueira 198220 NA NA Tongue NA NA NA Yasuoka 198448 40 M Tongue NA NA NA Segal 198436 NA NA Tongue NA NA NA NA NA Tongue NA NA NA Sambo 198633 NA NA Tongue NA NA NA Ling 198623 NA NA Tongue NA NA NA
Van der Wal 198742 61 F Tongue NA NA NA
Aguirre 19881 53 M Tongue NA NA NA Tani 198941 NA NA Tongue NA NA NA Ishibashi 198919 79 F Tongue NA NA NA Yamanaka 198947 20 F Tongue NA 1.50 NA Sanchez-Aniceto 199034 61 M Tongue NA NA NA Munro 199024 NA NA Tongue NA NA NA
Kostopoulos 199322 32 M Parotid gland NA NA S-100+
Blum 19934 60 F Cheek 1 year 1 NA
Kamysz 199621 5 F Neck 9 months NA NA
Wang 199845 20 M Parapharyngeal space 2 years NA NA
Sera 200537 17 M Tongue NA NA NA
Onodera 200526 47 F Cheek NA 4x2x2 S-100+, vim+
Aslam 20063 47 F Parotid gland 2 years 4x3x2 S-100+, vim+
De Riu 200710 47 M Masticatory space NA 3.5 S-100+, vim+
Vazquez Mahia200743 54 F Preauricular region 4 years 2.5x2.5 NA
Scivetti 200835 51 F Tongue NA 0.5 S-100+
Present case 49 M Masseter muscle 6 years 4 S-100+
directed against glial fibrillary acidic protein, cytokeratins, S-100 protein and CD-572,25.
To the authors’ knowledge, this is the first case of primary intramuscular chon-droma arising in the masseter muscle. In two comprehensive reports of extraosseus chondroma the predominant location of this tumour was in the hands and feet8,9. A review of the literature on head and neck soft tissue chondromas (Table 1) disclosed 34 cases of soft tissue chondro-mas occurring in the head and neck region, of which 24 were in the tongue1,3,11,14,19– 20,23–24,28,31–37,41–42,44,47,49–50
, 4 in the cheek4,16,18,26, 2 in the parotid gland3,22, 1 in the parapharyngeal space45, 1 in the preauricular region43, 1 in the neck21and 1 in the masticatory space10. The age range of the patients was 5–79 years. Soft tissue chondroma is more common in females than in males (even though the patient’s gender was not available in 16 cases) and the most common site is the tongue. The lesion has a slow and indolent course and occasionally is present for many years16. The treatment of choice is a wide local excision owing to the high recurrence rate of 10–15%15.
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Address: Luigi Insabato
Department of Biomorphological and Functional Sciences Pathology Section
University of Naples ‘‘Federico II’’, via S. Pansini 5 80131 Naples Italy Tel.: +39 0817463442 Fax: +39 0817463475 E-mail:[email protected]
